Recurrent sepsis in a patient with biliary hamartomas
  • Maria Beatriz Santos
    Internal Medicine Department, Hospital de Santo Espírito da Ilha Terceira, Angra do Heroísmo, Portugal
  • Magda Ponta Garça
    Internal Medicine Department, Hospital de Santo Espírito da Ilha Terceira, Angra do Heroísmo, Portugal https://orcid.org/0000-0002-1762-9302
  • Bárbara Vieira
    General Surgery Department Hospital de Santo Espírito da Ilha Terceira, Angra do Heroísmo, Portugal https://orcid.org/0000-0001-8267-8162
  • Paulo Ávila
    Internal Medicine Department, Hospital de Santo Espírito da Ilha Terceira, Angra do Heroísmo, Portugal https://orcid.org/0000-0002-0648-219X
  • Alexandra Freitas
    Internal Medicine Department, Hospital de Santo Espírito da Ilha Terceira, Angra do Heroísmo, Portugal

Keywords

Biliary hamartoma, von Meyenburg complex, sepsis, hepatic lesions

Abstract

Biliary hamartomas or von Meyenburg complexes (VMCs) are hepatic tumour-like lesions related to congenital malformation of the ductal plate, and are part of the ciliopathy spectrum of disorders. The exact pathogenesis of VMCs is unclear and it remains controversial whether they have the potential for malignant transformation. Patients are often asymptomatic and VMCs are usually encountered as an incidental finding on imaging.
We report a case of recurrent sepsis with an unidentified focus. It was later confirmed that biliary hamartomas were acting as a sanctuary for the persistent pathogenic agent. The authors hope to draw attention to the existence of this unusual focus of recurrent sepsis.

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References

  • Koay DSC, Leow WQ, Tan TT, Slapak G. Recurrent hepatobiliary sepsis in a patient with von Meyenburg complexes: a case report and review of the literature. Proceedings of Singapore Healthcare 2017;26:133–136.
  • Jáquez-Quintana JO, Reyes-Cabello EA, Bosques-Padilla FJ. Multiple biliary hamartomas, the “von Meyenburg complexes”. Ann Hepatol 2017;16:812–813.
  • Shin YM. Biliary hamartoma presented as a single mass. Korean J Hepatol 2011;17:331–334.
  • Zheng RQ, Zhang B, Kudo M, Onda H, Inoue T. Imaging findings of biliary hamartomas. World J Gastroenterol 2005;11:6354–6359.
  • Panda N, Brackett D, Eymard C, Kawai T, Markmann J, Kotton CN, et al. Liver transplantation for recurrent cholangitis from von Meyenburg complexes. Transplant Direct 2019;5:e428.
  • Barboi OB, Moisii LG, Albu-Soda A, Ciortescu I, Drug V. Biliary hamartoma. Clujul Med 2013;86:383–384.
  • Nagano Y, Matsuo K, Gorai K, Sugimori K, Kunisaki C. Bile duct hamartomas (von Mayenburg complexes) mimicking liver metastases from bile duct cancer: MRC findings. World J Gastroenterol 2006;12:1321–1323.
  • Hashimoto M, Ouchi M, Norose J, Futami-Suda S, Suzuki K, Matsumura N, et al. Bile duct hamartomas (von Meyenburg complexes) associated with a bacterial infection: case report of elderly diabetic patient. Geriatr Gerontol Int 2011;11:534–536.
  • Jain D, Sarode VR, Abdul-Karim FW, Homer R, Robert ME. Evidence for the neoplastic transformation of von-Meyenburg complexes. Am J Surg Pathol 2000;24:1131–1139.
  • Heinke T, Pellacani LB, de Olivera Costa H, Fuziy RA, Franco M. Hepatocellular carcinoma in association with bile duct hamartomas: report on 2 cases and review of the literature. Ann Diagn Pathol 2008;12:208–211.
  • Xu AM, Xian ZH, Zhang SH, Chen XF. Intrahepatic cholangiocarcinoma arising in multiple bile duct hamartomas: report of two cases and review of the literature. Eur J Gastroenterol Hepatol 2009;21:580–584.
  • Song JS, Noh SJ, Cho BH, Moon WS. Multicystic biliary hamartoma of the liver. Korean J Pathol 2013;47:275–278.
  • Sinakos E, Papalavrentios L, Chourmouzi D. The clinical presentation of von Meyenburg complexes. Hippokratia 2011;15:170–173.
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    Published: 2023-03-18
    Issue: 2023: Vol 10 No 3 (view)


    How to cite:
    1.
    Santos MB, Ponta Garça M, Vieira B, Ávila P, Freitas A. Recurrent sepsis in a patient with biliary hamartomas. EJCRIM 2023;10 doi:10.12890/2023_003764.

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